Shin Dong-Whan, Kim Ji-Eun, Kim Hee-Sun, Lee Ho
a Department of Oral and Maxillofacial Surgery, Section of Dentistry , Seoul Metropolitan Government - Seoul National University Boramae Medical Center , Seoul , Republic of Korea.
b Department of Pathology , Seoul National University College of Medicine , Seoul , Republic of Korea.
Cranio. 2017 Jan;35(1):58-63. doi: 10.1080/08869634.2016.1163807. Epub 2016 Apr 4.
Jacob's disease (JD) is an uncommon disorder that presents as pseudojoint formation between the enlarged mandibular coronoid process and the zygoma, resulting in mouth opening limitation (MOL). Intraosseous lipoma (IOL) usually presents in the long bone, but it is very rare in the mandible. The authors present a rare case of JD and coexisting IOL in the same anatomical area and discuss diagnosis and surgical intervention.
The patient was a 39-year-old Korean female with MOL. Magnetic resonance imaging showed a large lobulated mass with fat-specific signal intensity. A combined extraoral and intraoral approach was used to resect the large mass. Histopathologic examination revealed a degenerated lipomatous tumor replacing the marrow cavity, with cartilaginous cap formation on the articular surface.
Postoperatively, the patient showed uneventful healing and resolution of MOL.
雅各布病(JD)是一种罕见疾病,表现为下颌骨冠状突增大与颧骨之间形成假关节,导致张口受限(MOL)。骨内脂肪瘤(IOL)通常出现在长骨中,但在下颌骨中非常罕见。作者报告了一例罕见的同一解剖区域同时存在JD和IOL的病例,并讨论了诊断和手术干预。
患者为一名39岁的韩国女性,有张口受限症状。磁共振成像显示一个具有脂肪特异性信号强度的大分叶状肿块。采用口外和口内联合入路切除该大肿块。组织病理学检查显示一个退化的脂肪瘤样肿瘤取代了骨髓腔,关节表面有软骨帽形成。
术后,患者愈合顺利,张口受限症状得到缓解。